Diagnostic accuracy of imaging approaches for early tumor detection in children with Li-Fraumeni syndrome.
Pediatr Radiol
; 52(7): 1283-1295, 2022 06.
Article
em En
| MEDLINE
| ID: mdl-35391548
ABSTRACT
BACKGROUND:
The Toronto protocol for cancer surveillance in children with Li-Fraumeni syndrome has been adopted worldwide.OBJECTIVE:
To assess the diagnostic accuracy of the imaging used in this protocol. MATERIALS ANDMETHODS:
We conducted a blinded retrospective review of imaging modalities in 31 pediatric patients. We compared imaging findings with the reference standards, which consisted of (1) histopathological diagnosis, (2) corresponding dedicated imaging or subsequent surveillance imaging or (3) clinical outcomes. We individually analyzed each modality's diagnostic performance for cancer detection and assessed it on a per-study basis for chest and abdominal regional whole-body MRI (n=115 each), brain MRI (n=101) and abdominal/pelvic US (n=292), and on a per-lesion basis for skeleton/soft tissues on whole-body MRI (n=140).RESULTS:
Of 763 studies/lesions, approximately 80% had reference standards that identified 4 (0.7%) true-positive, 523 (85.3%) true-negative, 5 (0.8%) false-positive, 3 (0.5%) false-negative and 78 (12.7%) indeterminate results. There were 3 true-positives on whole-body MRI and 1 true-positive on brain MRI as well as 3 false-negatives on whole-body MRI. Sensitivities and specificities of tumor diagnosis using a worst-case scenario analysis were, respectively, 40.0% (95% confidence interval [CI] 7.3%, 83.0%) and 38.2% (95% CI 29.2%, 48.0%) for skeleton/soft tissues on whole-body MRI; sensitivity non-available and 97.8% (95% CI 91.4%, 99.6%) for chest regional whole-body MRI; 100.0% (95% CI 5.5%, 100.0%) and 96.8% (95% CI 90.2%, 99.2%) for abdominal regional whole-body MRI; sensitivity non-available and 98.3% (95% CI 95.3, 99.4) for abdominal/pelvic US; and 50.0% (95% CI 2.7%, 97.3%) and 93.8% (95% CI 85.6%, 97.7%) for brain MRI.CONCLUSION:
Considerations for optimizing imaging protocol, defining criteria for abnormalities, developing a structured reporting system, and practicing consensus double-reading may enhance the diagnostic accuracy for tumor surveillance.Palavras-chave
Texto completo:
1
Coleções:
01-internacional
Base de dados:
MEDLINE
Tipo de estudo:
Diagnostic_studies
/
Guideline
/
Prognostic_studies
/
Screening_studies
Limite:
Child
/
Humans
Idioma:
En
Ano de publicação:
2022
Tipo de documento:
Article